Bratati Dey et al. / International Journal Of Advances In Case Reports, 2021;8(1):27-30.

INTERNATIONAL JOURNAL OF e - ISSN - 2349 - 8005 ADVANCES IN CASE REPORTS

www.mcmed.us/journal/ijacr Case Report CYSTIC HYGROMA OF – A CASE REPORT AND LITERATURE REVIEW

1 2 3 4 5 Bratati Dey *, Savita Ghom , Anil Govindrao Ghom , Ajit Mishra , Shweta Singh

1PG student, Department of Oral and , Maitri College of and Research Centre, Anjora, Durg, Chhattisgarh 491001, India. 2Reader, Department of and Radiology, Maitri College of Dentistry and Research Centre, Anjora, Durg, Chhattisgarh 491001, India. 3Professor & HOD, Department of Oral Medicine and Radiology, Maitri College of Dentistry and Research Centre, Anjora, Durg, Chhattisgarh 491001, India. 4Professor, Department of Oral Medicine and Radiology, Maitri College of Dentistry and Research Centre, Anjora, Durg, Chhattisgarh 491001, India. 5Professor, Department of Oral Medicine and Radiology, Maitri College of Dentistry and Research Centre, Anjora, Durg, Chhattisgarh 491001, India.

ABSTRACT Cystic hygroma usually present as a painless swelling of the neck involving both the anterior and posterior triangle, with 44% in anterior triangle and 56% in posterior triangle. It is benign congenital tumour of neck region found mostly in children, but it can occur in any age. This is a rare case report of a child in which successful surgical removal of the cystic lesion with preservation of vital structure of the neck was performed. A discussion and brief literature review of cystic hygroma in child is described.

Key words: Cystic Hygroma; Painless; Benign; Congenital; Surgical Removal.

Corresponding Author: Bratati Dey Email: [email protected]

Access this article online Home page: Quick Response code http://www.mcmed.us/journal/ijacr

Received: 12.04.2021 Revised: 22.05.2021 Accepted: 15.06.2021

INTRODUCTION Hygroma is a Greek word which means ‘water- CASE REPORT containing tumour’.[1] Cystic Hygroma is a developmental A 8 years old girl weight of 26 kg, height of 4.5”, abnormality in which progressive dilatation of lymphatic resident of manki, presented to the Jeevan Rekha channels found resulting in lymphatic fluid accumulation. Multispecialty Hospital, Manki, Rajnandgaon, with the The term cystic hygroma is also known as lymphatic chief complaints of painless mass in the right side of neck malformation or or hydrocele of neck. It is since childhood, when her parents noticed a small swelling often present at birth and most cases are diagnosed in on the left side of neck, As the child growing the mass was infancy (<2 years).[2] increasing in size gradually. She also gave history of Cystic hygroma could classify into separated fullness of neck on left side and some discomfort on moving (multiloculated) or non-separated single cavity (non- her neck to downward. loculated). The incidence of cystic hygroma is very rare and There was no history of pain, dyspnea and is estimated to be 1 case per 6000-16000 births.[3] dysphasia, hoarseness of voice, low grade fever or loss of

27 | P a g e

Bratati Dey et al. / International Journal Of Advances In Case Reports, 2021;8(1):27-30. weight. The swelling did not move during deglutination. right side to posterior triangle of neck of left side. There was Patient gave no history of trauma in past in the respected no lymphadenopathy present and systemic examination was area. There was no history of bleeding or pus discharge, no normal. Her face was bilaterally symmetrical. history of increasing in size of the swelling during meal. Routine blood investigations were normal. X-Ray She was non-diabetic, non-hypertensive, no history of chest (PA) view showed prominent bronco vascular thyroidism, no history of previous hospitalization and no marking? Bronchitis. Fine needle aspiration cytology history of drug . Her past dental history and family (FNAC) revealed straw coloured fluid and it shows history was not relevant. microscopically (cytology) lymphocytes, which suggest Patient was examined on a sitting position under chronic inflammation. the broad daylight after taking an inform consent. She was Ultrasonographic evaluation of neck revealed conscious allert, co-operative and well oriented to the time evidence of large well defined cystic lesion of place person. She was nutritionally well hydrated with pulse approximately size 77×43mm with Interval low-level of 88beats/minute in the right radial artery which was echoes seen in left lateral aspect of neck suggestive of large regular in rhythm normal volume in character. Blood cystic mass? Cystic hygroma. CT-Scan of neck revealed pressure was 110/76 mmhg in the left arm supine position. large well-defined unilocular hypo dense cystic lesion in left Respiratory rate was 17 breaths/minute, regular thoraco- anterior neck space lateral to carotid trachea and thyroid, abdominal. posterior and medial sternocledomastoid muscle with No signs of cynosis, pallor, icturus, clubbing or contralateral deviation of the trachea and thyroid most likely pedal . Examination of the cranial nerve was normal. suggestive of unilocular cystic hygroma and the patient was The trachea was centrally placed with a visible mass in the scheduled for surgical excision. left side of neck. Under general anesthesia through a MacFee There was a cervical swelling present in the neck incision (fig.3.) full thickness flap was raised and region, which was well defined oval shaped mass measuring sternocledomastoid muscle was separated (fig.4.), the around 8.5-6 cm2 in the left side of the neck involving branches of spinal accessory nerve was seen over the cystic anterior triangle of neck. Colour of the mass was normal to lesion and common carotid artery and internal jugular vein the anatomic site. There was no erythema, sinus, scar or were carefully preserved (fig.7) and the cystic lesion was fistula present. Submandibular duct opening was visualized excised (fig.6.) and drainage placed, and suture done. Post with normal salivary flow of the mouth. operatively the patient recovered uneventfully, with On palpation the mass was soft, fluctuant and non- minimal drain the output which was removed on the 5th day; tender. Extension of the mass was supero-inferiorly, from on 9th day suture removal done. No recurrence found up to transverse process of atlas to supra clavicular fossa and 2 years of and patient was asymptomatic. antero-posteriorly thyroid gland and paratracheal region or

Fig. 1. An 8-year-old female presenting with a large left- Fig.2.1. At the level of hyoid bone sided neck swelling.

28 | P a g e

Bratati Dey et al. / International Journal Of Advances In Case Reports, 2021;8(1):27-30.

Fig.2.2. At the level of carotid artery Fig.2.3. At the level of clavicle

Fig.3. MacFee Fig.4. Fig.5. was Fig.6. Cystic Lesion Fig.7. Spinal accessory incision Sternocledomastoid completely detached nerve and brachial muscle separated. from its base plexus carefully preserved

DISCUSSION dimensional view, relation to adjacent structures and helpful It is believed that Cystic hygroma arises from a in surgical treatment planning.[5] Treatment options consist congenital malformation of the in which of surgery, laser excision, cauterization, , a failure of communication between the venous pathways simple drainage, radiation and radio-frequency ablation.[9] and lymphatic leads to fluid accumulation.[4] Cystic However, surgery remains the preferred modality of hygroma can occurs in any region of the body, the cervico- treatment.[6] The postoperative complications usually facial region (75-80%) is the common location followed by observed after surgical removal of the cystic lesion, they are axilla, mediastinum and groin.[3,5] For investigations of wound hemorrhage or lymphatic discharge, numbness of the cystic hygroma ultrasonography (USG) and CECT scan and neck and shoulder, infection, hypertrophied scar from the MRI can be used. Contrast enhanced computed tomography wound.[5] In cystic hygroma recurrence rate is high due to (CECT) scan can be describe the extent of the lesion in three incomplete excision of lesion.

29 | P a g e

Bratati Dey et al. / International Journal Of Advances In Case Reports, 2021;8(1):27-30.

Painless swelling present since birth with onset cystic hygroma: a case report and review of transillumination test positive may give clue to the diagnosis management. Rohit shrivastava et al.[9] (2014) reported a of cystic hygroma. In laboratory finding the cystic lesion difficult case of cystic hygroma. Paulo Rocha Pereira et consist of dilated cystic space lined by endothelial cells. In al.[2] (2015) reported cystic hygroma – a rare appearance in the present case the swelling present since childhood and adulthood. which is also painless. Tran illumination test reveals positive. Sometimes, overlying skin may be blue, whether CONCLUSION in the present case the colour of the swelling was normal to The etiology of cystic hygroma is unknown, the adjacent anatomic site. although it is believed that it is associated with chromosomal In old literature, V Ramchandra et al.[7] (2001) reported anomalies in 60-75% of the cases. Cervical recurrent cystic hygroma. And P Saxena et al.[8] (2009) may be isolated or can associate with syndromes; they are reported a case report on cystic hygroma of neck in an adult the , the Klippel-Trenauny syndrome, the female. Furthermore L. Gow et al. (2011)[4] describe adult congenital glaucoma.[10]

REFERENCES 1. Rajendra K. Ghritlaharey, Management of Giant Cystic Lymphangioma in an Infant Journal of Clinical and Diagnostic Research. 2013, 7(8): 1755-6. 2. Paulo Rocha Pereira, Tatiana Carvalho, Marta Canas Marques, Ana Rita Santos, Antonio Marques Pereira Cystic Hygroma - A Rare Appearance in Adulthood Open Journal of clinical and medical 2015; 1(6). 3. Kamble V et al.Cystic hygroma with : a rare case report. Int J Reprod Contracept Obstet Gynecol. 2014;3(3):847-850 4. L. Gow et al. Adult-onset cystic hygroma: a case report and review of management Grand Rounds. 2011;1:5–11. 5. Seffah JD , Adu-Bonsaffoh k. giant recurrent cystic hygroma: a case report journal of the west african college of surgeons 2014;4(2) 6. Marcelo Gadelha Vasconcelos Oral lymphangioma: case report RSBO. 2011;8(3):352-6. 7. V Ramachandran, P Rajan. Recurrent Cystic Hygroma. The Internet Journal of Surgery. 2001; 3(1). 8. P Saxena, D Chandra. Cystic Hygroma of neck in an adult female: Case report and literature review. The Internet Journal of Surgery. 2009; 22(2). 9. Srivastava R et al. A difficult case of cystic hygroma Int Surg J.2014;1(3):167-9 10. Sarafoleanu et al. The diagnosis of a cervical tumefaction in children. Romanian Journal of Rhinology, 2015;5(17).

Cite this article: Bratati Dey, Savita Ghom, Anil Govindrao Ghom, Ajit Mishra, Shweta Singh. Cystic Hygroma Of Neck – A Case Report And Literature Review. International Journal of Advances In Case Reports, 8(1), 2021, 27-30.

Attribution - NonCommercial - NoDerivatives 4.0 International

30 | P a g e